Investigations of morphological changes in photoreceptors in Behçet's disease (BD) using adaptive optics scanning light ophthalmoscope (AOSLO) are lacking. The authors aimed to evaluate macular cone density and regularity in BD patients with or without a history of uveitis with good visual acuity (VA). The authors included 16 patients (29 eyes) with BD and 12 healthy volunteers (12 eyes) as controls. All subjects had VA of 20/20 or higher. Subjects underwent AOSLO to evaluate the photoreceptor status including cone density (numbers/mm2) and proportion of hexagonal Voronoi domains. Hyporeflective patches that were not detected in color fundus photograph or optical coherence tomography were observed by AOSLO in BD patients both with and without past uveitis history; these were not detected in the control group. Cone density was significantly reduced in BD patients with a history of uveitis compared to controls (P = .002). The proportion of hexagonal Voronoi domains was significantly reduced in BD eyes both with and without history of uveitis relative to controls (P < .001). Macular photoreceptor damage was observed in BD patients with and without a history of uveitis. [Ophthalmic Surg Lasers Imaging Retina. 2021;52:218-225.].